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Series of Laryngomalacia, Tracheomalacia, and Bronchomalacia Disorders and Their Associations with Other Conditions in Children

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Series of Laryngomalacia, Tracheomalacia, and Bronchomalacia Disorders and Their Associations with Other Conditions in Children Pediatric Pulmonology 34:189-195 (2002) Series of Laryngomalacia, Tracheomalacia, and Bronchomalacia Disorders and Their Associations With Other Conditions in Children I.B. Masters, MBBS, FRACP,1* A.B. Chang, PhD, FRACP,2 L. Patterson, MBBS, FANZCAC,1 С Wainwright, MD, FRACP,1 H. Buntain, MBBS,1 B.W. Dean, MSC,1 and P.W. Francis, MD, FRACP1 Summary. Laryngomalacia, bronchomalacia, and tracheomalacia are commonly seen in pediatric respiratory medicine, yet their patterns and associations with other conditions are not well-understood. We prospectively video-recorded bronchoscopic data and clinical information from referred patients over a 10-year period and defined aspects of interrelationships and associations. Two hundred and ninety-nine cases of malacia disorders (34%) were observed in 885 bronchoscopic procedures. Cough, wheeze, stridor, and radiological changes were the most common symptoms and signs. The lesions were most often found in males (2:1) and on the left side (1.6:1). Concomitant malacia lesions ranged from 24%forlaryngotracheobronchomalaciato 47% for tracheobronchomalacia. The lesions were found in association with other disorders such as congenital heart disorders (13.7%), tracheo-esophageal fistula (9.6%), and various syndromes (8%). Even though the understanding of these disorders is in its infancy, pediatricians should maintain a level of awareness for malacia lesions and consider the possibility of multiple lesions being present, even when one symptom predominates or occurs alone. Pediatr Pulmonol Pediatr Pulmonol. 2002; 34:189-195. © 2002 wiiey-Liss. inc. Key words: laryngomalacia; tracheomalacia; bronchomalacia; malacia disorders; syndromes. INTRODUCTION The aim of this report is to describe an extensive experience of various forms of laryngomalacia, tracheo­ Tracheomalacia, bronchomalacia, and laryngomalacia malacia, and bronchomalacia and explore some of the disorders are commonly seen in tertiary pediatric respira­ interrelationships that exist between these conditions with tory practice. These disorders of the large airways are respect to their anatomical sites and associations, associated with cough, wheeze, and stridor, covering most of the common respiratory symptoms seen in children. The importance of diagnosing tracheomalacia, broncho­ METHODS malacia, and laryngomalacia ranges from life-threatening A detailed review of the prospectively recorded cli­ disease requiring major diagnostic and surgical interven­ nical features and videotaped bronchoscopic lesions of tions to parental reassurance and withdrawal of drugs the airways from children referred to the authors over a 7 often used to treat these disorders unnecessarily.' ~ In our 10-year period was undertaken. The children were collective experience, many children are only referred for referred to the Royal Children's Hospital and Mater assessment after repeated failures of trials of asthma drug therapies and antibiotics. Department of Respiratory Medicine. Royal Children's Hospital, Herston. However, with the symptoms involved often over­ Brisbane. Queensland, Australia. lapping other respiratory disorders, it is not surprising that the management of these disorders could be viewed as "Flinders University Northern Territory Clinical School, /Mice Springs suboptimal. In addition, the prevalence of these disorders Hospital, Alice Springs, Australia. is not known, and just what relationships exist between "Correspondence to: Dr. I.B. Masters. MBBS, FRACP, Department of these disorders and vascular and cardiovascular disor­ Respiratory Medicine, Royal Children's Hospital. Hcrston. Brisbane 4029. ders, immunological disorders, recurrent bronchitis, bron­ Queensland. Australia. E-mail: [email protected] chiolitis, asthma, chronic suppurative lung disease and pneumonia are also not known, making decisions of Received 27 November 200 I; Accepted 23 May 2002. management even more difficult in terms of diagnostic DOI l0.U)02/ppuU0!5o precision, drug therapies, and long-term prognostication, Published online in Wiley InterScience Avww.intcrscience.wilev.com) © 2002 Wiley-Liss, Inc. 190 Masters et al. Children's Hospital, Brisbane, from throughout the state the bronchoscope or applied positive airway pressures. of Queensland, Australia, where the total population is The usual appearance of malacia in the left main stem approximately 3 million, of whom 500,000 are children. was that of an "inverted teardrop," with partial or These referral centers handle almost all complex pedi­ complete apposition of the mucosal superior and inferior atric respiratory medicine for the state. surfaces producing a slit-like opening (Fig. 9a,b). Lobar Children with the endoscopic diagnosis of laryngoma- or segmental lesions usually had slit-like or "lunar lacia, bronchomalacia, or tracheomalacia were selected crescent appearances" (Fig. 10a,b). from all bronchoscopic procedures performed. The endoscopic diagnostic features are outlined below, Materials and Bronchoscopic Procedure and examples are given in Figures 1-10. For the purposes of this report, we introduce the term "malacia disorders" All diagnostic information was obtained during flexible to cover the lesions as a group. bronchoscopic procedures performed during spontaneous breathing under gaseous general anesthesia. The anes­ Definition 1 thetic agents used were halothane and or sevoflurane. The vocal cords and trachea were sprayed with lignocaine Laryngomalacia is a long, curved epiglottis, short ary- from a Cass needle or direct spray. Generally, the children epiglottic folds (false cords), prominent and or bulky breathed spontaneously throughout the procedure, and arytenoids and degrees of prolapse of the arytenoids, and positive end expiratory pressure was not usually applied changes in epiglottic appearance and position seen during except during periods of assessment of malacia. The inspiration and returning to the original position during bronchoscope was passed into the airway through a port expiration. in the connector to the facemask. The same four anesthetists were involved in all procedures. All cases Definition 2 were videotaped for later perusal. Anatomical sites and Tracheomalacia is a membranosa deformity in the type of lesion were detailed in each case. All bron­ trachea, maintained during spontaneous respiration but choscopic procedures were performed when the children which could be altered by the passage of the broncho­ were clinically well or stable. A Pentax FB10X 3.5 mm scope or positive airway pressure changes. The descrip­ or Olympus BF 3C40 3.6 mm bronchoscope was used in tive appearances accepted as tracheomalacia are as each case. follows: triangular (Fig. 1), oval/flattening (Fig. 2a-c), localized extrathoracic raised and dynamically bulging Statistics pars membranosa (tracheal dyskinesia) (Fig. 3a,b), and an "Eifel Tower" appearance (Fig. 4a,b). All data were expressed as simple percentages, medi­ Pericarinal tracheobronchomalacia was defined as a ans, and ranges. flattening in the anterior/posterior plane of the trachea, extending into the left and right main-stem bronchi RESULTS (Figs. 5a,b, 6a-c). Tracheomalacia associated with tracheo-esophageal fistula is usually seen as a combina­ Two hundred and ninety-nine cases of laryngomalacia, tion of a dynamically bulging pars membranosa and tracheomalacia, bronchomalacia, and/or combinations of anterior flattening or triangular appearances (Figs. 7a- the three were defined from a total of 885 bronchoscopies d, 8a-c). performed over a 10-year period. This represents 34% of the procedures being defined a "malacia disorder." Definition 3 Malacia disorders were defined in 205 males and 94 females, i.e., a male to female ratio of 2:1. In recognized Bronchomalacia is an appearance of deformity in the syndromes and related disorders, the male predominance large right or left main-stem bronchi, and/or their res­ ranged from 1.6:1 to 8:1. The ratios and associated pective divisions at the lobar or segmental level. The conditions are shown in Tables 1 and 2. appearance of deformity could be altered by a passage of The median age of this cohort was 9 months (range, 1 day to 240 months), with the median age of males being ABBREVIATIONS 9 months, and that of females being 9.6 months. Stridor was reported in 127 (42.5%), and of these, 58 CLE Congenital lobar emphysema CT Cono-truncal (19.4%) reported stridor alone. Wheeze and cough LMS Left main .stem symptomatology was reported in 130 (43.4%) and 212 RMS Right main stem (71%), respectively, while 34 (11.7%) reported all three SYN Syndrome symptoms. These signs were not usually present in very TEF Tracheo-esophageal lislula sick patients, particularly cardiac patients, where unex- Malacia Disorders in Children 191 Fig. 1. Triangular appearance of tracheomalacia. Fig. 4. "Eifel tower" appearance of tracheomalacia (a), and progressive expiratory closure with mucosal surfaces apposed (b). Fig. 5. Pericarinal right main-stem malacia (a), and progressive closure of right main stem on expiration (b). Fig. 2. Pericarinal and right and left main-stem malacia. Oval shape (a) and progressive flattening on expiration of pericarinal and right and left main-stem bronchi (b), and complete closure of L main stem bronchi (c). r Fig. 3. Bulging pars membranosa: subglottis and proximal trachea: "posterior tracheomalacia." Dynamically bulging pars Fig. 6. Pericarinal and left main-stem malacia (a),and progres­ starting in the subglottis (a), and progressing to less prominent sive closure of left main
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